Subretinal Fibrosis Developing 10 Years After First Presentation with Chronic Ocular Sarcoidosis in a Child.

IF 2.6 4区 医学 Q2 OPHTHALMOLOGY
Ocular Immunology and Inflammation Pub Date : 2025-01-01 Epub Date: 2024-06-12 DOI:10.1080/09273948.2024.2363481
Neofytos Mavris, Tarek Jaouni, Radgonde Amer
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引用次数: 0

Abstract

Purpose: To provide a comprehensive overview of the diagnostic and therapeutic journey of a pediatric patient with persistent sarcoid-associated panuveitis over a 10-year period, who ultimately developed bilateral macular subretinal fibrosis and visual loss.

Methods: Retrospective case report.

Results: The patient was diagnosed with sarcoidosis after undergoing a transbronchial biopsy. She was followed up because of granulomatous panuveitis, multifocal choroiditis, and papillitis bilaterally. She maintained a stable condition, and visual acuity was 0.3 RE and 0.5 LE. Immunomodulatory therapy included prednisone, methotrexate, and adalimumab. The patient was lost to follow-up for 20 months because of the COVID-19 pandemic. She was represented with active uveitis and was not responding to TNF-ɑ inhibitors (adalimumab and infliximab). Ultimately, the patient's intraocular inflammation was successfully controlled by using intravitreal steroids (Triamcinolone and Fluocinolone acetonide implant). However, the visual outcome was guarded because of bilateral subretinal fibrosis.

Conclusion: 10% of patients with sarcoidosis-associated uveitis risk blindness in one eye. The index case progressed to sight-robbing bilateral subretinal fibrosis, a rare complication of ocular sarcoidosis despite a combination of conventional and biologic anti-inflammatory therapies. There is a pressing need to develop new treatment agents for refractory non-infectious uveitis.

一名儿童首次患上慢性眼肉样瘤病 10 年后出现视网膜下纤维化。
目的:全面概述一名患有持续性肉样瘤相关泛葡萄膜炎的儿科患者的诊断和治疗历程,该患者在10年时间里最终发展为双侧黄斑视网膜下纤维化和视力丧失:方法:回顾性病例报告:结果:患者在接受经支气管活检后被诊断为肉样瘤病。由于出现肉芽肿性泛葡萄膜炎、多灶性脉络膜炎和双侧乳头炎,她接受了随访。她的病情保持稳定,视力为 0.3 RE 和 0.5 LE。免疫调节治疗包括泼尼松、甲氨蝶呤和阿达木单抗。由于 COVID-19 大流行,患者失去了 20 个月的随访。她被诊断为活动性葡萄膜炎,对 TNF-ɑ 抑制剂(阿达木单抗和英夫利昔单抗)无效。最终,患者通过使用玻璃体内类固醇(曲安奈德和氟西酮醋酸酯植入剂)成功控制了眼内炎症。结论:10%的肉样瘤病相关葡萄膜炎患者有单眼失明的风险。结论:10%的肉样瘤病相关葡萄膜炎患者有单眼失明的风险,该病例的病情恶化为双侧视网膜下纤维化,这是一种罕见的眼部肉样瘤病并发症,尽管已采用常规和生物抗炎疗法。目前迫切需要开发治疗难治性非感染性葡萄膜炎的新药。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
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来源期刊
CiteScore
6.20
自引率
15.20%
发文量
285
审稿时长
6-12 weeks
期刊介绍: Ocular Immunology & Inflammation ranks 18 out of 59 in the Ophthalmology Category.Ocular Immunology and Inflammation is a peer-reviewed, scientific publication that welcomes the submission of original, previously unpublished manuscripts directed to ophthalmologists and vision scientists. Published bimonthly, the journal provides an international medium for basic and clinical research reports on the ocular inflammatory response and its control by the immune system. The journal publishes original research papers, case reports, reviews, letters to the editor, meeting abstracts, and invited editorials.
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